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It is now clear that both CD4+ and CD8+ subsets contain CD45RA+ memory cells.. Over that period a vast literature has described the phenotype, location and behaviour of T cells derived f

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β 2 m = β 2 microglobulin; IFN- γ = interferon-γ; IL = interleukin; LFA-1 = lymphocyte function-associated antigen-1; NK = natural killer cells; PPD = purified protein derivative; RA = rheumatoid arthritis; SCID = severe combined immunodeficiency; SF = synovial fluid; TCR = T cell receptor; Th = T helper cells; TNF = tumour necrosis factor; TREC = T cell receptor excision circle.

Introduction

Despite many years of study, the aetiology of inflammatory

arthritis remains poorly understood A growing body of

data describing leukocyte differentiation, migration and

cellular interactions has put us in a promising position to

further dissect the molecular basis of inflammatory

arthritis A recent meeting brought together more than 60

researchers from across the UK at the Kennedy Institute of

Rheumatology, Imperial College, London The informal

atmosphere of the meeting encouraged the presentation

of recent results and novel ideas by 20 speakers covering

four themes

T cell activation and differentiation

Professor M Salmon (Birmingham University, UK) outlined

recent changes in the model of T cell differentiation in

which activation turns naive CD45RA+ T cells into CD45RO+primed/memory T cells, which divide periodically until they die It is now clear that both CD4+ and CD8+

subsets contain CD45RA+ memory cells Detailed study

of CD8 memory using MHC class I/viral peptide tetramers has defined several new models of CD8 differentiation according to the changing expression of numerous cell surface markers Memory CD45RA+cells are now widely accepted; their function, particularly proliferative potential,

is currently under debate Professor Salmon showed proliferation in CD8CD45RA+ memory cells, but only under stringent stimulation conditions; this may explain the poor responses reported for these cells These new concepts of differentiation have prompted re-examination

of T cells in arthritis Lymphocyte function-associated antigen-1 (LFA-1) and the chemokine receptor CCR7

Meeting report

Kennedy Institute of Rheumatology Division, Imperial College

London, 12–13 November 2003: Towards a molecular toolkit for

studying lymphocyte function in inflammatory arthritis

Jeff Faint1 and Frances Hall2

1 The University of Birmingham/MRC Centre for Immune Regulation, University of Birmingham, Birmingham, UK

2 University of Cambridge School of Clinical Medicine, Addenbrooke’s Hospital, Cambridge, UK

Corresponding author: Jeff Faint (e-mail: j.m.faint@bham.ac.uk)

Received: 10 Feb 2004 Revisions requested: 11 Feb 2004 Revisions received: 18 Feb 2004 Accepted: 19 Feb 2004 Published: 8 Mar 2004

Arthritis Res Ther 2004, 6:55-59 (DOI 10.1186/ar1162)

© 2004 BioMed Central Ltd (Print ISSN 1478-6354; Online ISSN 1478-6362)

Abstract

T lymphocytes have been implicated in the pathogenesis of inflammatory arthritis for approximately

30 years Over that period a vast literature has described the phenotype, location and behaviour of

T cells derived from patients with inflammatory rheumatological disease The arthritiogenic roles of

MHC class I and class II molecules, which present antigen to T cells, have been hotly debated The T cell

has been variously conceived as a central or peripheral (or even incidental) component in the

arthritogenic response Rapid developments in genomics and use of biological therapeutic agents

coupled with recent insights in the field of T cell differentiation and immunoregulation together offer

novel methods of investigating the pathogenesis of chronic inflammatory disease A number of UK

researchers, with diverse interests within the field of synovitis, met recently at the Kennedy Institute of

Rheumatology Presentations on T cell memory, cytokines of homeostasis and inflammation,

unconventional behaviour of MHC molecules and immunoregulation in murine models, rheumatoid and

spondyloarthritis reflected the breadth of the discussion

Keywords: cytokines, HLA-B27, immunoregulation, migration, rheumatoid arthritis, spondyloarthritis

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discriminate the two CD45RA+ populations in healthy

subjects; naive cells are LFA-1lowCCR7high, memory cells

LFA-1highCCR7low [1] Dr J Faint (Birmingham University,

UK) has characterised CD8+CD45RA+ cells found in

rheumatoid synovial infiltrates Synovial CD8CD45RA+

cells are LFA-1high memory cells, containing Epstein–Barr

virus tetramer binding cells in seropositive subjects Some

synovial, but not blood, CD8CD45RA+ memory cells

expressed CCR7, which could be induced by culture in

rheumatoid synovial fluid (SF) CCR7 directs migration to

lymph nodes, with naive T cells migrating through high

endothelial venules, and maturing tissue dendritic cells to

afferent lymphatics These data suggest that tissue

infiltrating T cells might operate a similar mechanism to

return to draining lymph nodes

T cell differentiation in arthritis was also examined by Dr F

Ponchel (Leeds University, UK), using differential

expres-sion of CD45 isoforms and T cell receptor exciexpres-sion circle

(TREC) analysis [2] TRECs are not replicated during

division and provide an indication of the replicative history

of cell populations Patients with rheumatoid arthritis (RA)

had reduced frequencies of naive and ‘conventional’

memory cells compared with healthy donors, yet expressed

additional populations not evident in controls This might

result from lymphopoenia, which is a feature common to

many diseases Reduced bone marrow stromal cell

production of interleukin (IL)-7 in rheumatoid patients

leads to a lack of circulating cytokine, which was restored

in some patients by therapy with anti-tumour necrosis

factor-α (anti-TNF-α) antibodies

In addition to the alterations in subset frequencies, T cells

in rheumatoid patients are hyporesponsive to stimulation

through the T-cell receptor (TCR) Dr A Cope (Kennedy

Institute, Imperial College, London, UK) demonstrated that

TCR triggering leads to transient internalisation and

subsequent re-expression of TCR/CD3 Chronically

stimulated cells, particularly in the presence of TNF-α,

show sustained low-level expression of the ζ signalling

chain of the CD3 complex, impairing signal transduction in

these cells [3] TCRζdimcells express many markers typical

of highly differentiated, senescent effector cells, and

respond poorly to stimulation by CD3/CD28 The

rheumatoid synovium is highly enriched in TCRζdim cells,

which might explain their hyporesponsiveness, while also

suggesting that effector responses of these cells are

relatively independent of antigen signals Tissue-infiltrating

cells in arthritis seem to be chronically stimulated, yet it is

unclear how they compare with cells during an evolving

immune response Professor A Akbar (University College

London, UK) has modelled a cutaneous inflammatory

response to purified protein derivative (PPD), showing the

T cell infiltrate to be oligoclonal and extensively

proliferating; 80% of clones were maintained between

days 7 and 19 of the response T cell proliferation is

affected by telomere shortening (repeating sequences of DNA found at the chromosome ends) with each cell division, eventually leading to replicative senescence; the enzyme telomerase can also lengthen telomeres Shortened telomeres were seen from day 14, yet little telomerase

activity was evident in vivo Cells stimulated in vitro

upregulated telomerase This was inhibited by fluid obtained from inflammatory blisters and might explain the limitation of expansion of specific T cells at sites of inflammation Synovial infiltrating T cells have been extensively studied, yet they are far outnumbered by neutrophils Professor R Moots (Liverpool University, UK) has been dissecting the interactions between these cells Joint neutrophils are long lived and produce many inflammatory mediators Neutrophils from healthy subjects express MHC class II after culture in a cytokine cocktail, whereas rheumatoid joint neutrophils spontaneously express MHC class II after overnight culture [4] These neutrophils can present

superantigen to T cells in vitro, suggesting a possible

antigen-presenting function for neutrophils; however, this

activity has yet to be demonstrated in vivo.

Lymphocyte recognition of HLA-B27

The association of ankylosing spondylitis with HLA-B27 is among the strongest described for an HLA locus, giving

an odds ratio of about 161 HLA-B27 also exhibits associations with other spondyloarthropathies, including reactive arthritis, psoriatic spondyloarthritis and spondylo-arthritis associated with inflammatory bowel disease

Dr P Bowness (Weatherall Institute of Molecular Medicine, Oxford, UK) discussed the unusual biology of HLA-B27 molecules and the implications of this for recognition by T cells and natural killer (NK) cells Classically the heavy chain of MHC class I molecules folds

in association with β2microglobulin (β2m) and a nonamer peptide This monomer presents the peptide to TCRs on CD8+T cells Indeed, the HLA-B27 monomer is capable

of presenting several peptides derived from bacterial triggers of reactive arthritis HLA-B27 is unusually (although not uniquely) able to form heavy-chain homodimers, via a disulphide bridge at Cys 67 [5] Modelling this complex suggests that the α1 helix unfolds and that the peptide-binding groove can accommodate a peptide much longer than a nonamer Expression of HLA-B27 heavy chain in cells deficient in TAP (transporter associated with antigen processing) results in homodimer formation, demonstrated

by binding of homodimer-specific monoclonal antibody Mice transgenic for HLA-B27, but deficient for β2m, express large quantities of surface homodimer HLA-B27+

β2m+mice express the conventional monomeric molecule but low levels of homodimer are also detectable HLA-B27 homodimer tetramers have revealed that about 5% of peripheral CD8+ T cells are capable of recognising the HLA-B27 in this conformation Of great interest is the

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observation that homodimer tetramers also bind to B cells,

monocytes and NK cells Further work is under way to

characterise the profile of receptors that recognise the

homodimer, and the relationship of homodimer formation

to the development and activity of spondyloarthritis

Conventionally, HLA-B27/peptide is recognised by CD8+

T cells The hypothesis that some CD4+ T cells might

recognise HLA-B27 arose from observations in

spondylo-arthritic HLA-B27-transgenic mice [6] Despite the apparent

importance of HLA-B27 in the pathogenesis, CD8+T cells

were shown not to be essential for the development of

arthritis; indeed, disease was transferable with CD4+T cells

Professor JSH Gaston (University of Cambridge School of

Clinical Medicine, UK) discussed the characterisation of a

variety of unconventional T cell clones from patients with

spondyloarthritis Several patterns of specificity in these

CD4+clones have emerged: first, proliferation in response

to HLA-B27, either empty monomers or, possibly,

homo-dimers; second, proliferation in response to an

HLA-B27-derived peptide presented by HLA-Cw1; and third,

proliferation in response to an HLA-B27-derived peptide

presented by alloantigens, including HLA-B51 and HLA-A2

Dr H Bodmer (Edward Jenner Institute for Vaccine Research,

Newbury, UK) continued the discussion on the nature of

HLA-B27-restricted CD4+ T cells A double transgenic

mouse model expressing GRb (TCR specific for HLA-B27

presenting influenza nucleoprotein 383–391) and HLA-B27/

β2m revealed the presence of HLA-B27-restricted CD4+

as well as CD8+ T cells The CD4+ cell required the

presence of MHC class II to be selected, presumably by

dual α-chain expression [7] However, the selected CD4+

T cells responded to HLA-B27/NP(383–391) with similar

sensitivity to the CD8+T cells In vitro, both Th1 and Th2

differentiation of HLA-B27-restricted CD4+ T cells could

be achieved The expression of MHC class II molecules is

generally restricted to professional antigen-presenting

cells, so as to regulate the initiation of responses by CD4+

T helper cells Recognition of MHC class I molecules by

CD4+T cells could therefore have important implications

for the initiation of autoimmunity Professor Gaston also

discussed the isolation of unconventional CD8+ T cells

from patients with spondyloarthropathy These autoreactive

CD8+T cell lines/clones produce IL-4 but not interferon-γ

(IFN-γ) They constitutively express the αβ TCR, CD69,

CD25 and CD30 and express CD40L on activation; they

are perforin-negative and express only low levels of

granzyme These clones are restricted by autologous

MHC class I molecules and are dependent on an

unidentified (ubiquitous?) peptide The possibility that they

are CD8+T regulatory cells is under investigation

Immunoregulation

Immunoregulation is an integral part of normal innate and

adaptive immune responses Recently, the literature has

burgeoned with the reports of the regulatory properties of CD4+T cells which constitutively express CD25 It is clear that regulation does not reside exclusively within this group but has also been attributed to subsets of CD8+

T cells (see above), dendritic cells, NK cells, neutrophils and eosinophils, as well as to the plethora of soluble regulatory molecules A tangential but germane observation is that pathogenic microbes have developed

an astonishing capacity for host immunointerference Many multicellular parasites exemplify the delicate balance of immunomodulation without global immunosuppression; this permits chronic infection of the host, often over decades

Dr L Taams (Kings College, London, UK) presented comparisons of the phenotype and function of CD4+

CD25+T cells in healthy controls and in both peripheral

blood and SF of patients with RA In vitro, the

CD4+CD25+T cells from RA and control peripheral blood were equally potent at inhibiting the proliferation of CD4+CD25− T cells The CD4+CD25+ T cells derived from RA SF were more potent suppressors but, curiously, the CD4+CD25−T cells from SF were more resistant to proliferative suppression Peripheral blood CD4+CD25+

cells modified cytokine release in an antigen-specific stimulation of CD4+CD25− cells TNF-α production was reduced in CD4+CD25− cells from peripheral blood, whereas IFN-γ and IL-10 were both decreased in CD25−

cells from SF Continuing the theme of regulatory T cells in

RA, Michael Ehrenstein (Centre for Rheumatology Research, London, UK) discussed the change in function

of CD4+CD25+T cells in the peripheral blood of patients with RA before and after TNF-α blocking therapy In comparison with healthy controls, patients with RA demonstrated a defect in the ability of their CD4+CD25+

T cells to inhibit the production of TNF-α by monocytes This defect was unchanged by treatment with methotrexate but was restored in patients who were successfully treated with TNF-α blockade

Dr L Wedderburn (Institute of Child Health, London, UK) advanced the hypothesis that immune regulation contributes

to the mild phenotype of children with oligoarticular juvenile idiopathic arthritis compared with patients with extended oligoarthritis The CD25+ and particularly CD25hiT cells in the SF of patients with juvenile idiopathic arthritis exhibited a regulatory phenotype (GITR+, CTLA-4+, [FoxP3 mRNA]high) The regulatory activity of this subset was also supported by the demonstration that depletion of

SF CD25+cells releases the proliferation of CD25−cells

T cells in SF from children with oligoarthritis expressed an approximately ten-fold higher CD25 expression than T cells from patients with extended oligoarthritis, which is of particular interest because the most potent regulatory activity has been shown to reside in the CD4+CD25hi

subset Children with mild disease also had higher overall numbers of CD4+CD25+ cells in the SF Although

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suppressive potency per cell seemed comparable in cells

from patients with mild and severe disease, these

observations are consistent with the hypothesis that the

CD4+CD25+ regulatory subset is less effective in the

children who develop extended oligoarticular arthritis

Frances Hall (University of Cambridge School of Clinical

Medicine, UK) presented data from a murine model of

inflammatory arthritis that suggested a role for regulatory

T cells in the repression of arthritis and dermatitis Adult

DBA/1 males were thymectomised and then treated with

CD25-depleting antibody, which depleted both

CD4+CD25med and CD4+CD25hi cells Although a low

level of inflammatory arthritis is usually evident in elderly

DBA/1 males, the CD25-depleted mice developed more

severe arthritis Histologically, this was associated with

pannus formation and erosions In addition, about 60% of

the CD25-depleted mice also developed an ulcerating

rash, characterised by a dense neutrophilic infiltrate The

nature of the emerging inflammatory disease and the

influence of the genetic background of the mice are

currently under investigation

Dr L Pazmany (Clinical Sciences Centre, Liverpool, UK)

broadened the immunoregulatory discussion by considering

the role of NK cells NK cells are present at autoimmune

sites of pathology, including the rheumatoid joint [8] In the

murine model of extrinsic allergic encephalitis, depletion of

NK cells resulted in an exacerbation of disease For other

models of autoimmune disease, results of NK depletion

have been variable The effect of addition of autologous

NK cells to a PPD-pulsed culture of T cells and

autolo-gous antigen-presenting cells was investigated, using

proliferation as a read-out The effect of freshly isolated

NK cells was dependent on the donor However, NK cells

incubated with IL-12 reproducibly inhibited T cell

proliferation, even at NK:T ratios of 1:10 The significance

of these observations for the susceptibility of individuals to

RA and for the severity of disease is being investigated

Professor I McInnes (University of Glasgow, UK)

demonstrated how the immunoregulatory prowess of

multicellular parasites might inform our search for the

optimal disease-modifying agent The filarial protein ES62

promotes a Th2 response in the BALB/c mouse It seems

to bind to TLR4 and promotes the development of type 2

dendritic cells, thereby decreasing IL-12 and TNF-α

production by macrophages In the murine

collagen-induced arthritis model, serial subcutaneous ES62

administration decreases the severity, although not the

incidence, of disease [9] This is associated with a

decrease in the proliferation and production of IL-6, IFN-γ

and TNF-α by draining lymph node cells from bovine

collagen type II immunised mice This effect of ES62 is

mirrored by a decrease in the production of TNF-α and IL-6

in primary synovial membrane cultures Because parasitic

products such as ES62 might be well tolerated for decades, without global immunosuppression, they seem a promising therapeutic strategy for Th1-dominant inflammatory diseases, such as RA

Homing and effector function

Tissue-specific ‘address codes’ have been defined for skin and gut homing lymphocytes by using the pattern of chemokine receptors and adhesion molecules that they express Isolating specific microvascular vessels has proved difficult and has prevented the definition of a synovial tissue code Professor C Pitzalis (Guy’s Hospital, London, UK) outlined a novel approach to this problem by using phage display libraries constructed from blood vessels formed in human synovial tissue transplanted into SCID (severe combined immunodeficiency) mice [10] Initial results show promise in isolating joint-specific homing markers He also showed that the rheumatoid joint has a similar spatial organisation of lymphocytes and chemo-kines to that seen in lymph nodes Lymph node develop-ment requires careful and coordinated interactions between different cell types; these results indicate that similar inter-actions might occur in the joint The theme of cell–cell interactions was continued by Professor F Brennan (Kennedy Institute, Imperial College, London, UK) T cells prolong spontaneous TNF-α release from cultured rheuma-toid synovial membrane cells through cell contact with

macrophages Using an in vitro model, T cells activated by

TCR crosslinking induced monocytes to produce TNF-α and IL-10, whereas T cells stimulated with a cytokine cocktail (analogous to bystander activation) induced predominantly TNF-α production By using pharmaco-logical inhibitors it now seems that these distinct responses arise through the recruitment of different signal transduction pathways in responder macrophages Thus, phosphoinositide 3-kinase inhibitors reduced cytokine production induced by TCR-stimulated T cells, yet NFκB inhibitors enhanced the response The opposite responses

to inhibitors were seen if bystander T cells were used Importantly, rheumatoid synovial T cells behave like bystander activated cells, providing further clues to the mechanisms of T cell effector responses in the joint CD3− CD56+NK cells present in bystander activated populations also induced TNF-α from synovial membrane cells in a contact-dependent manner Joint-infiltrating CD56+ cells were shown by Professor M Callan (Imperial College, London, UK) to enhance TNF-α production by monocytes About 10% of NK cells from peripheral blood express higher levels of CD56 than the majority of cells, yet CD56bright cells were the predominant population infil-trating the joint [11] Attempts to differentiate CD56intinto CD56brightcells have failed, indicating that they represent two separate lineages CD56brightNK cells express low levels of perforin, mount poor cytotoxic responses and produce many proinflammatory cytokines on stimulation These activities might further exacerbate responses in inflammatory sites

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Professor J Isaacs (Newcastle University, UK) concluded

with some words of caution about our ability to develop

novel antibody therapies, which have generally proved less

effective in humans than in the mouse This might be due

to targeting the wrong molecules or the wrong epitopes of

those molecules, or to incorrect doses and duration of

therapy Tolerance might take time to develop, and

concomitant therapy might actually hinder its development

A further complication is that some therapies might initially

exacerbate symptoms before providing benefits This might

partly account for the clinical improvement seen in some

patients in antibody trials after treatment was stopped

owing to disease flares The challenge for the future will lie

in recognising what are side effects of treatment and what

are potentially dangerous turns in disease

Conclusions

The overwhelming message from this meeting was the

appreciation of not only the diversity of cell types present

in the inflamed joint but also the diversity of their

interactions No cell type seems to be solely responsible

for the maintenance of inflammation; rather, it is the

interactions between the multiple cell types present This

presents a number of potential targets for future therapies,

yet suggests that an effective cure will require multiple

interventions targeting multiple pathways

Competing interests

None declared

References

1 Faint JM, Annels NE, Curnow SJ, Sheilds P, Pilling D, Hislop AD,

Wu L, Akbar AN, Buckley CD, Moss PAH, Adams DH, Rickinson

AB, Salmon M: Memory T cells constitute a subset of the

human CD8+CD45RA+ pool with distinct phenotypic and

migratory characteristics J Immunol 2001, 167:212-220.

2 Ponchel F, Morgan AW, Bingham SJ, Quinn M, Buch M, Verburg

RJ, Henwood J, Douglas SH, Masurel A, Conaghan P, Gesinde M,

Taylor J, Markham AF, Emery P, van Laar JM, Isaacs JD:

Dysregu-lated lymphocyte proliferation and differentiation in patients

with rheumatoid arthritis Blood 2002, 100:4550-4556.

3 Isomaki P, Panesar M, Annenkov A, Clark JM, Foxwell BM,

Cher-najovsky Y, Cope AP: Prolonged exposure of T cells to TNF

down-regulates TCR zeta and expression of the TCR/CD3

complex at the cell surface J Immunol 2001, 166:5495-5507.

4 Cross A, Bucknall RC, Cassatella MA, Edwards SW, Moots RJ:

Synovial fluid neutrophils transcribe and express class II

major histocompatibility complex molecules in rheumatoid

arthritis Arthritis Rheum 2003, 48:2796-2806.

5 Bird LA, Peh CA, Kollnberger S, Elliott T, McMichael AJ, Bowness

P: Lymphoblastoid cells express HLA-B27 homodimers both

intracellularly and at the cell surface following endosomal

recycling Eur J Immunol 2003, 33:748-759.

6. Boyle LH, Goodall JC, Opat SS, Gaston JS: The recognition of

HLA-B27 by human CD4 + T lymphocytes J Immunol 2001,

167:2619-2624.

7 Roddis M, Carter RW Sun MY, Weissensteiner T, McMichael AJ,

Bowness P, Bodmer HC: Fully functional HLA-B27-restricted

CD4+ as well as CD8+ T cell responses in TCR transgenic

mice J Immunol 2004, 172:155-161.

8 Pridgeon C, Lennon GP, Pazmany L, Thompson RN, Christmas SE,

Moots RJ: Natural killer cells in the synovial fluid of rheumatoid

arthritis patients exhibit a

CD56bright,CD94bright,CD158nega-tive phenotype Rheumatology 2003, 42:870-878.

9 McInnes IB, Leung BP, Harnett M, Gracie JA, Liew FY, Harnett W:

A novel therapeutic approach targeting articular inflammation

using the filarial nematode-derived

phosphorylcholine-containing glycoprotein ES-62 J Immunol 2003,

171:2127-2133.

10 Lee L, Buckley C, Blades MC, Panayi G, George AJ, Pitzalis C:

Identification of synovium-specific homing peptides by in vivo

phage display selection Arthritis Rheum 2002, 46:2109-2120.

11 Dalbeth N, Callan MF: A subset of natural killer cells is greatly

expanded within inflamed joints Arthritis Rheum 2002, 46:

1763-1772.

Correspondence

Jeff Faint, Rheumatology Unit, Division of Immunity and Infection, The University of Birmingham, Edgbaston, Birmingham B15 2TT, UK Tel: +44 121 415 8690; fax: +44 121 414 3599; e-mail: j.m.faint@bham.ac.uk

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